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Journal of Korean Association of Pediatric Surgeons 7

pISSN 2383-5036 eISSN 2383-5508J Korean Assoc Pediatr Surg Vol. 21, No. 1, June 2015http://dx.doi.org/10.13029/jkaps.2015.21.1.7 Case Report

Periappendiceal Actinomycosis Presenting as Acute Appendicitis

Jung-Hak Kwak1, Eu-Jin Won1, Eun-Hwa Choi2, Sung-Eun Jung1, Hyun-Young Kim1

Departments of 1Pediatric Surgery and 2Pediatrics, Seoul National University Childrens Hospital, Seoul, Korea

Abdominal actinomycosis is a rare and chronic progressive disease, especially in children. Clinically, it has non-specific symptoms and diagnostic findings as well as low prevalence, making it very difficult to diagnose prior to intraoperative pathological confirmation. For this reason, abdominal actinomycosis is commonly misdiagnosed as appendicitis. After the histopathological diagnosis of abdominal actinomycosis is made, patients should be administered an appropriate antibiotic such as penicillin. Here we describe a case of appendi-ceal actinomycosis in an 18-year-old girl who was initially diagnosed with acute appendicitis.

Keywords: Actinomycosis, Appendiceal neoplasms, Appendiceal actinomycosis

Received: November 11, 2014, Revised: January 23, 2015, Accepted: March 9, 2015

Correspondence: Hyun-Young Kim, Department of Pediatric Surgery, Seoul National University Childrens Hospital, 101 Daehak-ro, Jongno-gu, Seoul 110-744, Korea.Tel: +82-2-2072-2478, Fax: +82-2-747-5130, E-mail: [email protected]

Copyright 2015 Korean Association of Pediatric Surgeons. All right reserved.This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

INTRODUCTION

Actinomycosis is a relatively rare and chronic pro-

gressive suppurative disease that is mainly caused by

Actinomyces israelii. A. israelii is an anaerobic Gram-

positive organism that is normally present in the oral

cavity as well as throughout the gastrointestinal tract,

female genital tract, and the bronchus [1] that spreads by

direct extension across the tissue planes with the for-

mation of multiple abscesses, abundant granulation tis-

sue, and draining sinuses [2]. Actinomycosis occurs most

frequently in the cervical facial (50% to 65%), abdominal

(20%), and thoracic (15%) regions [1].

Abdominal actinomycosis, the incidence of which has

increased over the past few years [3], usually involves the

vermiform appendix and terminal ileum [3,4]. Its clinical

symptoms are non-specific and include abdominal pain,

fever, nausea, vomiting, and diarrhea [3]. If the appendix

ruptures, right-sided pain and other symptoms of appen-

dicitis can develop [3-5]. A combination of antibiotics

and surgical intervention is the treatment of choice for

abdominal actinomycosis [6].

Here we describe an 18-year-old girl who underwent

a laparoscopic exploration for acute appendicitis and was

diagnosed with appendiceal actinomycosis upon histo-

pathological examination.

CASE REPORT

An 18-year-old girl who had a 4-day history of right

lower abdominal pain visited at a nearby hospital, and

she had undergone laparoscopic exploration for a pre-

sumed diagnosis of acute appendicitis. At that hospital,

an unexpected mass-like lesion beneath the cecum in the

right lower quadrant (RLQ) was found, and she was

transferred to our hospital for further evaluation.

Physical examination revealed tenderness in the RLQ

and no rebound pain. There were no associated symptoms

such as fever, loss of appetite, nausea or vomiting, or

diarrhea. She had no relevant past or family history.

Laboratory findings included mild leukocytosis

(11,760/L; neutrophils 79.7%, eosinophils 0.8%) and an elevated C-reactive protein level (3.09 mg/dL).

A contrast-enhanced abdominal CT scan revealed a

4.32.3 cm mass-like lesion beneath the cecum in the

RLQ and an invisible normal appendix (Fig. 1).

From these findings, acute appendicitis with a peri-

toneal abscess was primarily diagnosed and a laparo-

8 Journal of Korean Association of Pediatric Surgeons

J Korean Assoc Pediatr Surg 2015;21(1):7-10

Fig. 1. CT image shows a 4.32.3 cm mass-like lesion beneath the cecum within the right lower quadrant (arrow). This finding suggested

differential diagnosis of advanced appendicitis with phlegmon or a

tumorous condition.

Fig. 2. Intraoperative findings. (A) Lapar-oscopic appearance of appendiceal ac-

tinomycosis. (B) Gross appearance of

appendiceal actinomycosis; a 7.04.0cm appendiceal mass. Severe inflam-

mation and adhesion between the ap-

pendix and the adjacent appendiceal serosal surface is visible.

Fig. 3. (A) Gross appearance of the excised specimen. (B) Pathological examination: Presence of Actinomyces israelii. Acute suppurative appendicitiswith a periappendiceal abscess and actinomycotic colonies characterized by sulfur granules (H&E stain, 40). (C) Pathological examination: Presence of A. israelii. Acute suppurative appendicitis with a periappendiceal abscess and actinomycotic colonies characterized by sulfur granules (H&E stain, 200).

scopic exploration was planned.

Operative findings showed a 33 cm appendiceal mass

with dense severe adhesions (Fig. 2A, B) for which we

performed an ileocecectomy.

Gross pathology revealed severe inflammation and

adhesion between the appendix and the adjacent appen-

diceal serosal surface. Irregular nodularity was seen in

the distal portion of the ileal mucosal area, while intra-

luminal obliteration was found due to the severe in-

flammation (Fig. 3A).

Light microscopic examination revealed acute suppu-

rative appendicitis with a periappendiceal abscess and

actinomycotic colonies. At higher magnification, a typi-

cal sulfur granule surrounded by neutrophils was found

that was confirmed as appendiceal actinomycosis (Fig.

3B, C).

After the operation, she was administered intravenous

Journal of Korean Association of Pediatric Surgeons 9

Kwak JH, et al: Periappendiceal Actinomycosis

antibiotics (cefotaxime, metronidazole, piperacillin/ta-

zobactam) for 20 days and then oral amoxicillin 1 g three

times a day for at least 3 months.

DISCUSSION

Abdominal actinomycosis is a rare and insidious dis-

ease [7,8]. The overall incidence of actinomycosis is vir-

tually impossible to ascertain since failure to consider it

as a diagnosis and difficulty confirming it lead to un-

der-reporting [9]. Approximately 65% of abdominal ac-

tinomycosis cases occur in association with acute appen-

dicitis [10].

The symptoms, signs, and laboratory findings of ab-

dominal actinomycosis are nonspecific, while its radio-

logic findings often show a poorly defined mass with ir-

regular margins [11]. As such, its diagnosis is often

delayed. Only 10% of cases are diagnosed preoperatively,

and it is difficult to differentiate abdominal actino-

mycosis from a malignant tumor or appendicitis [7].

The differential diagnosis of abdominal actinomycosis

should include Crohns disease, ulcerative colitis, diver-ticulitis, neoplasms, and intestinal tuberculosis [8,11].

The definitive diagnosis of actinomycosis requires mi-

croscopic proof of either the pathogen itself or the pres-

ence of specific sulfur granules [8].Tissue sampling and a histologic examination are im-

portant to ensuring a correct diagnosis, but the former is

not always possible for two reasons. First, the clinical

presentation of the disease may be acute or the sample

may be unobtainable using percutaneous methods. Second,

Actinomyces is a Gram-positive, slow-growing, non-

acid-fast, anaerobic, and filamentous bacterium that re-

quires specific incubation in special culture medium for

at least 7 days [1]. Accordingly, culture results are neg-

ative in 76% of cases [12] and excision biopsy for histo-

pathological examination is frequently needed [1].

The combined use of surgery and penicillin therapy

has a reported cure rate of 88.8% [13]. Antibiotic therapy

is useful for preventing the local recurrence of actino-

mycosis. Penicillin in particular has been shown to be ef-

fective in treating abdominal actinomycosis. Other com-

monly used antibiotics include tetracycline, erythromycin,

doxycycline, clindamycin, imipenem, ceftriaxone, and

ciprofloxacin [7].

A combination of long-term antibiotic therapy and

adequate surgery is necessary to ensure the complete

eradication of actinomycosis because of the large degree

of reactive fibrosis formed by the infection [14]. Howev-

er, recent studies have shown that a combination of com-

plete surgical resection followed by short-term anti-

biotic treatment is effective [15].

Similarly, in this case, as the disease does not present

with any typical features, either clinically or radio-

logically, the patient was initially diag